|Year : 2015 | Volume
| Issue : 1 | Page : 131-135
Central ossifying fibroma of mandible: A case report and review of literature
Anand N Swami, Lata M Kale, Sunil Surendraprasad Mishra, Sneha H Choudhary
Department of Oral Medicine and Radiology, Chhatrapati Shahu Maharaj Shikshan Sanstha (CSMSS) Dental College, Aurangabad, Maharashtra, India
|Date of Submission||03-Jun-2014|
|Date of Acceptance||14-Sep-2015|
|Date of Web Publication||12-Oct-2015|
Sunil Surendraprasad Mishra
Department of Oral Medicine and Radiology, Chhatrapati Shahu Maharaj Shikshan Sanstha (CSMSS) Dental College, Aurangabad, Maharashtra
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Ossifying fibroma (OF) is a benign, non-odontogenic tumor of the jaw, a type of fibro-osseous lesion. Traditionally, this type of lesion was subclassified histologically into ossifying fibroma and cementifying fibroma according to the hard tissues formed, but both types are now known by the unified term, ossifying fibroma. It is generally accepted that the histological subclassification of these two lesions is of academic interest only since differential diagnosis is often arbitrary and their biological behaviour seems to be identical. The present article discusses the case of central ossifying fibroma in a 35-year-old female patient who presented with a swelling in premolar-molar region of left mandible which was symptom-free and present since last 6 months. The diagnosis was confirmed by histopathology.
Keywords: CT scan, fibro-osseous lesions, mandible, ossifying fibroma
|How to cite this article:|
Swami AN, Kale LM, Mishra SS, Choudhary SH. Central ossifying fibroma of mandible: A case report and review of literature. J Indian Acad Oral Med Radiol 2015;27:131-5
|How to cite this URL:|
Swami AN, Kale LM, Mishra SS, Choudhary SH. Central ossifying fibroma of mandible: A case report and review of literature. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2020 Jun 1];27:131-5. Available from: http://www.jiaomr.in/text.asp?2015/27/1/131/167134
| Introduction|| |
Ossifying fibroma is a benign fibro-osseous lesion that demonstrates a well-demarcated proliferation of cellular fibrous tissue with varying amounts of osseous products including bone, cementum or a mixture of both. , Although it has been categorized under fibro-osseous lesions including the orofacial region, it behaves like a benign bone neoplasm. WHO in 1972 classified it in two types as ossifying fibroma and cementifying fibromas; but in 1992 WHO considered it in one heading as cemento-ossifying fibroma. Further, the term "cementossifying fibroma" was replaced by "ossifying fibroma" in 2005 under the new WHO classification. ,, The central variant of ossifying fibroma is a relatively rare lesion which is commonly found in females than in males, and has a predilection for mandible than maxilla. From the radiological perspective, the lesion manifests as unilocular, well-demarcated, mixed radiolucent-radiopaque region based on the presence of cementum or bone.  The article reports a case of unilocular central ossifying fibroma of mandible in a 35-year-old female with a brief discussion on its literature.
| Case Report|| |
A 35-year-old woman was referred for evaluation of swelling in the left mandibular body [Figure 1]. The patient stated that the mass had been gradually increasing in size since 6 months. The past medical and dental histories were non-contributory. Also the family history was not contributory. Clinical examination revealed a small localized swelling measuring approximately 5 × 2 cm in size in the left body of mandible in premolar molar region. Swelling was bony hard in consistency and asymptomatic completely. On examination, no lymphadenopathy was detected. On intraoral examination obliteration of buccal vestibule in premolar molar region was noted [Figure 1]. No abnormality was detected in relation to the vitality of teeth in the affected area. There was no mobility or displacement of any teeth. Diagnostic guidelines including complete hemogram, serum calcium, serum phosphorus, serum alkaline phosphatase were suggestive of no abnormalities. A clinical diagnosis of benign odontogenic tumor was made.
In radiographic evaluation, mandibular occlusal cross-sectional view showed region of cortical expansion in the left premolar-molar region [Figure 2]. Orthopantomograph (OPG) revealed a mixed radiolucent-radiopaque lesion of size approximately 6 × 4 cm extending from 34 to 37 region and superiorly from apical portion of 36 and 37 to inferiorly upto the superior band of inferior border of mandible. The borders of the lesion were non-corticated, irregular but well defined and showed some amount of scalloping especially in the supero-lateral aspect [Figure 3]. Computed tomographic (CT) scan revealed heterodense lesion with irregular expansion of buccal and lingual cortical plates and with central foci of calcification [Figure 4] and [Figure 5]. These findings led to a radiographic diagnosis of ossifying fibroma. OPG and CT scan also revealed another single oval, ill-defined radiopaque lesion superimposed on the apical portion of 45 which was suggestive of cementoblastoma or idiopathic osteosclerosis.
|Figure 2: Mandibular occlusal cross-sectional view showing cortical expansion in left premolar-molar region|
Click here to view
|Figure 3: OPG view shows well-defined mixed radiopaque-radiolucent lesion with radiolucent band at periphery in 44 to 47 tooth region. Inferior border of the mandible is intact|
Click here to view
|Figure 4: 3D CT showing well-demarcated lesion with few radiopaque spicules on left side of mandible and an oval radiopaque lesion on right side (s/o cementoblastoma)|
Click here to view
|Figure 5: Axial CT showing expansion of cortices with central radiopaque foci|
Click here to view
With a working diagnosis of central ossifying fibroma, the patient was advised surgical excision of the lesion. The excised specimen was sent for histopathologic evaluation. Histological examination confirmed the diagnosis by revealing presence of immature bone trabeculae with entrapped osteocytes and lined by a dense rim of enlarged osteoblasts [Figure 6]. The patient is under follow-up for last 6 months, and no recurrence has been reported yet [Figure 7] and [Figure 8].
|Figure 6: Immature bone trabeculae with entrapped osteocytes and lined by a dense rim of enlarged osteoblasts (hematoxylin and eosin, x400)|
Click here to view
|Figure 7: Post-operative clinical picture taken after 6 months showing no evident clinical complications|
Click here to view
| Discussion|| |
The aetiology of ossifying fibroma is unknown but odontogenic, developmental and traumatic origins have been suggested and thought to be of periodontal ligament origin because of their capacity to produce cementum and osteoid material.  Ossifying fibroma develops from the multipotential mesenchymal cells of periodontal ligament origin which are able to form both bone and cementum. , Although the precise pathogenesis is still unknown, Wenig et al. , has suggested that trauma-induced stimulation may play a role. Recent genetic studies have revealed a mutation in tumor suppressor gene HRPT2, a protein product known as parafibronin which leads to tumor formation. Few studies have also reported chromosomal abnormalities such as translocation and interstial deletion of codings in chromosome 2. ,
Clinically, the cemento-ossifying fibroma presents as a painless, slowly growing mass in the jaw where displacement of teeth may be the only early clinical feature.  The tumor is well-circumscribed from its surrounding bone and will continue to grow bigger, slowly or actively, with larger lesions occasionally leading to facial deformity.  Tumor shows female preponderance with ratio of 5:1. Previous studies report an age range of 10-59 years; however, few others have concised this range to be 20-40 years. , In mandible its occurrence is 70-90%, where it occurs more frequently in premolar-molar region followed by the involvement of maxilla, ethmoidal and orbital regions also. Bilateral involvements has also been reported in few cases. ,,, In our case, we found similar features with the patient being a female in her fourth decade of life presenting with a painless swelling in the mandibular premolar region. Although swelling was present there was no displacement of the affected teeth.
Conventional radiographs and specialized imaging techniques such as CT and CBCT helps in differentiating this lesion from other fibro-osseous lesions. The lesions may be either unilocular or multilocular.  In the early stages, the ossifying fibroma appears as a radiolucent lesion with no evidence of internal radiopacities. With increasing tumor maturity radiographs show increase in the appearance of radiopaque masses which may coalesce to form a large radiopaque foci surrounded by radiolucent border. The ossifying fibroma presents a radiolucent appearance in 53%, a sclerotic radio density in 7% and mixed or mottled appearance in 40% of the cases.  An important diagnostic feature radiographically is that there is a centrifugal growth pattern rather than a linear one and therefore the lesions grow by centripetal direction and present as a round tumor mass.  Another important radiographic feature of COF which helps in distinguishing it from other benign fibro-osseous lesions is that it is more well circumscribed. There are three different patterns of radiographic borders of cemento-ossifying fibroma which are: Defined lesion without sclerotic border (40%); defined lesion with sclerotic border (45%) and lesion with ill-defined border (15%) indicating a rapidly growing tumor. 
Central ossifying fibromas are typically well-circumscribed, solitary radiolucencies with scattered radiopaque foci. The lesion commonly occurs at apices of vital teeth in premolar-molar region. There is spherical expansion without cortical perforation, and may lead to divergence of adjacent teeth.  In our case the lesion was spherical in shape and expansile but the lesion was not well-circumscribed as mentioned in literature time and again. Also there was no displacement of teeth. The nasal septum, infraorbital foramen and orbital floor may be involved, if the tumor is large. Surgical therapy is decided by the extent of tumor.
Histopathological features include proliferation of irregularly shaped calcifications within a hypercellular fibrous connective tissue stroma. The calcifications are extremely variable in appearance and represent various stages of bone and cementum deposition. Histologic differentiation between osteiod and cementum is difficult. Additional biochemical studies such as ultrastructural studies and polarized studies have been utilized for differentiating between cementum-like material and bone in these lesions with no definite results. 
Differential diagnoses include other mixed radiolucent radiopaque lesions such as fibrous dysplasia, periapical cemental dysplasia, condensing osteitis, odontoma; entities which may contain calcification foci like calcifying epithelial odontogenic cyst, calcifying epithelial odontogenic tumor, adenomatoid odontogenic tumor and osteogenic sarcoma. Ossifying fibroma has well-defined margins and is covered by a soft tissue capsule. Fibrous dysplasia has margins which blend with the surrounding bone. Ossifying fibroma shows a concentric expansion of bone around a definite epicentre which leads to alteration in bone morphology, displacement of teeth and also resorption of tooth root. On contrary to this, fibrous dysplasia causes minimum changes maintaining the normal bone morphology and rarely causes root resorption. The major differentiating point in distinguishing ossifying fibroma from periapical cemental dysplasia is the teeth vitality. Also periapical cemental dysplasia shows multifocal origin in contrast to ossifying fibroma. One or more missing teeth are associated with calcifying epithelial odontogenic cyst, calcifying epithelial odontogenic tumor or adenomatoid odontogenic tumor. There is also presence of multifoci calcification. In odontoma, pattern of calcification resembles that of tooth structure. Malignant features like ill-defined radiolucency, destructed bone with soft tissue involvement are suggestive of ossifying fibroma. ,
Ossifying fibromas comprises entities with different morphological features that can be mistaken for other benign fibro-osseous lesions; this similarity and overlapping microscopic characteristics turns the multidisciplinary approach, comprehending clinical, radiological and pathological aspects, more reliable for a correct diagnosis.  They have locally aggressive behavior, with high recurrence rate, particularly in partial and incomplete excisions, with complete removal being the gold standard treatment. Prognosis is good, without metastases in the reported cases. 
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Hamner JE 3 rd
, Scofield HH, Cornyn J. Benign fibro-osseous jaw lesions of periodontal membrane origin. An analysis of 249 cases. Cancer 1968;26:861-78.
Sarwar HG, Jindal MK, Ahmad SS. Cemento-ossifying fibroma - A rare case. J Indian Soc Pedod Prev Dent 2008;26:128-31.
Reichart PA, Philipsen HP, Sciubba JJ. The new classification of head and neck tumours [WHO]-any changes? Oral Oncol 2006;42:757-8.
Liu Y, Wang H, You M, Yang Z, Miao J, Shimizutani K, et al
. Ossifying fibromas of the jaw bone: 20 cases. Dentomaxillofac Radiol 2010;39:57-63.
Sreenivasan B, Edward JO, Sunil S. Central ossifying fibroma of mandible- A case report. Oral Maxillofac Path J 2010;1:Jan-Jun. [Last accessed on 2011 Oct 24].
Carvalho B, Pontes M, Garcia H, Linhares P, Vaz R. Ossifying Fibromas of the Craniofacial Skeleton. In: Poblet E, editor. Histopathology - Reviews and Recent Advances. InTech. Available from: http://www.intechopen.com/books/histopathology-reviews-and-recent-Advances/ossifying-fibromas-of-the-craniofacial-skeletonhttp://dx.doi.org/10.5772/51030. [Last accessed on 2014 May 03].
Bertolini F, Caradonna L, Bianchi B, Sesenna E. Multiple ossifying fibroma of the jaws: A case report. J Oral Maxillofac Surg 2002;60:225-9.
Regezi JA, Sciubba JJ. Oral Pathology: Clinico Pathologic Correlations. 3 rd
ed. Philadelphia: W.B. Saunders; 1999. p. 357-60.
Wenig BL, Sciubba JJ, Cohen A, Goldstein MN, Abramson AL. A destructive maxillary cemento-ossifying fibroma following maxillofacial trauma. Laryngoscope 1984;94:810-5.
Ong AH, Siar CH, Cemento-ossifying fibroma with mandibular fracture. Case report in a young patient. Aust Dent J 1998;43:229-33.
Neville BW, Damm DD, Allen CM, Bouquot JE. Bone pathology. In: Chi AC, editor. Oral and Maxillofacial Pathology. 3 rd
ed. Noida: Elsevier; 2009. p. 647.
Jayachandran S, Sachdeva S. Cemento-ossifying fibroma of mandible. Report of two cases. J Indian Acad Oral Med Radiol 2010;22:53-6.
Langlais RP, Langland OE, Notje CJ. Diagnostic Imaging of the Jaws. Baltimore: Williams & Wilkins; 1995. p. 551-2.
Barberi A, Cappabianca S, Colella G. Bilateral cemento-ossifying fibroma of the maxillary sinus. Br J Radiol 2003;76:279-80.
Kuta AJ, Worley CM, Kaugars GE. Central cemento ossifying fibroma of the maxillary sinus: A Review of six cases. AJNR Am J Neuroradiol 1995;16:1282-6.
Brademann G, Werner JA, Jänig U, Mehdorn HM, Rudert H. Cemento-ossifying fibroma of petromastoid region: Case report and review of the literature. J Laryngol Otol 1997;111;152-5.
Sanchis JM, Peñarrocha M, Balaguer JM, Camacho F. Fibroma cemento-osificante mandibular: Presentación de dos casos y revisión de la literatura. Med Oral Patol Oral Cir Bucal 2004;9: 69-73.
Sapp JP, Eversole LR, Wysocki GP. Contemporary Oral and Maxillofacial Pathology. 2 nd
ed. St Louis, Missouri: Mosby; 2004. p. 116-7.
Kerr AR, Phelan JA. Benign Lesions of the oral cavity. In: Greenberg MS, Glick M, Ship JA, editors. Burket's Oral Medicine. 11 th
ed. New Delhi: BC Decker Inc.; 2008. p. 143-5.
Ojo MA, Omoregie OF, Altini M, Coleman H. A clinic-pathologic review of 56 cases of ossifying fibroma of the jaws with emphasis on the histomorphologic variations. Niger J Clin Pract 2014; 17:619-23.
Wood NK, Goaz PW, Jacobs MC. Periapical radiolucencies. In: Wood Nk, Goaz PW, editors. Differential Diagnosis of Oral and Maxillofacial Lesions. 5 th
ed. New Delhi: Mosby; 2011. p. 266-7.
White SC, Pharoah MJ. Diseases of bone manifested in jaws. In: White SC, Pharoah MJ, editors. Oral Radiology: Principle and Interpretation. 6 th
ed. New Delhi: Mosby; 2004. p. 500-1.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]